CASE REPORT – OPEN ACCESS

International Journal of Surgery Case Reports 10 (2015) 52–55

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International Journal of Surgery Case Reports

journal homepage: www.casereports.com

A rare combination of hepatic and pericardial hydatid cyst and review of literature

Kallol Dasbaksi ∗, Suranjan Haldar, Kaushik Mukherjee, Plaban Mukherjee

Department of Cardiothoracic Surgery, Medical College Hospital, Kolkata, 88, College Street, Kolkata 700073, West Bengal, India

article info abstract

Article history: Hydatid disease in human beings, as in all intermediate hosts, manifest as hydatid cyst (HC). It is an Received 14 January 2015 important cyclozoonotic disease, endemic in various sheep and cattle raising areas of the world, including Accepted 28 February 2015 India. The tapeworm commonly involved is Echinococcus granulosus. HC can occur almost anywhere in the Available online 14 March 2015 body, most common organs being liver and lungs, and are usually solitary. In 25% of cases combination of liver HC with HC in other extra pulmonary locations are found. Cardiac HCs comprise of 0.5–2% of Keywords: all HC cases. Within the , HCs are usually situated in the left or right and rarely found in Hydatid cyst of liver the peri-. Pericardial HC does not produce symptoms and is often painless and silent, until the Hydatid cyst of cysts grow to a large size over the years, when the usual complications develop, such as cyst rupture, cardiac compression, atrial fibrillation, and even sudden death. We describe the case of a 39 year old house wife, of rural origin, with proximity to livestock, who had an asymptomatic pericardial HC along with a symptomatic hepatic HC. She clinically presented with an abdominal lump for one year with recent onset of abdominal pain for 1 month, when radiological imaging confirmed the diagnosis of an unruptured hepatic HC and a pericardial HC. The patient recovered after pericardiectomy along with excision of the HC over the left ventricle and enucleation of hepatic HC, by thoracoabdominal approach. She is doing well after 5 years of followup without recurrence. © 2015 Published by Elsevier Ltd. on behalf of Surgical Associates Ltd. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Case report chambers and effusion. The wall of the cardiac chambers were free of any space occupying lesion. Computerized tomogram (CT) of the A 39 year old lady, from a remote rural location of Gangetic Delta abdomen and thorax showed a 14 cm by 8 cm oval cyst in left lobe of South West Bengal, presented to us with gradually increasing of liver (Fig. 1C) and an elliptical cystic lesion of 12 cm by 5 cm in the swelling of the upper abdomen noted for 1 year and chronic abdom- pericardial cavity overlying the left ventricle (LV) (Fig. 1B and D). inal pain for 1 month. A large, mildly tender and non pulsatile Serology was positive for hydatid disease. There was eosinophilia, lump was present in the left upper quadrant of abdomen, moving but other routine investigations were normal. Preoperative diagno- with respiration without an upper palpable margin. She was hemo- sis was hydatid cyst (HC) of liver and pericardium. Oral albendazole dynamically stable without any respiratory distress. There was tablets 400 mg twice daily was started during the preoperative pallor, but no icterus, dependent edema, jugular venous engorge- period. ment and ascitis. Both heart and breath sounds were normal. Chest X-ray (Fig. 1A) showed an increased cardiothoracic ratio with a 2. Operation bulge along the left heart border and an area of increased opac- ity in left lower zone. Ultrasonograpy (USG) of abdomen revealed Exploration was done by a left thoracoabdominal approach a cystic lesion in the left lobe of liver and also suggested a cyst under general anesthesia. There was no pulmonary adhesion. The in the pericardium. Echocardiography confirmed the presence of pericardial hydatid cyst (PHC) was situated mainly over the left a localized cyst in the pericardial cavity over the left ventricle ventricle and partly over the right. The surrounding area was iso- without any pressure effect and communication to the ventricular lated with large swabs soaked with 5% povidone iodine solution which was also injected into the cyst. The parietal pericardium was thick and after incising the pericardium (Fig. 2A) approximately ∗ 500 cc of fluid was drained. The pericardium along with the con- Corresponding author at: 23, New Santoshpur 1st Lane, East Jadavpur, Kolkata 700075, India. Tel.: +91 9830321432. tents of HC, were dissected off the ventricular wall (Fig. 2B) and E-mail addresses: [email protected], [email protected] (K. Dasbaksi). excised, leaving behind a clear myocardial surface (Fig. 2C and D).

http://dx.doi.org/10.1016/j.ijscr.2015.02.052 2210-2612/© 2015 Published by Elsevier Ltd. on behalf of Surgical Associates Ltd. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). CASE REPORT – OPEN ACCESS

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Fig. 1. (A) Chest X-ray showing and increased cardiothoracic ratio with a bulging along the . (B) Shows longitudinal section of thoracic CT showing the pericardial cyst over the left heart border. (C) Shows the cyst in the left lobe of liver. (D) Shows the transverse section of the CT showing the elliptical pericardial hydatid cyst over the left ventricle.

Fig. 2. (A) Pericardial cyst being incised after injection of diluted 5% povidone iodine into the pericardial cystic cavity. Clear fluid under pressure being sucked out. (B) The thickened pericardium is being incised to expose the heart. The bands between the pericardium and the epicardium are shown, which are remnants of the smaller secondary hydatid cysts, and are being dissected and excised along with fibrous pericardium to expose the clean epicardial surface (shown by transparent white arrow in the figure C and D in the right hand corners). (C) Shows the liver cyst after clearance. (D) Shows omentum tuked into the liver cyst as shown by the black bordered white arrow. The divided costal arch (transparent black arrows in both C and D) with the attached diaphragm separating the liver and the heart shown, before thoracoabdominal wound closure. CASE REPORT – OPEN ACCESS

54 K. Dasbaksi et al. / International Journal of Surgery Case Reports 10 (2015) 52–55

The hepatic hydatid cyst (HHC) was enucleated with usual precau- explanation is, contiguous spread of infective protoscolices from a tions and the cavity was packed with omentum (Fig. 2C). Wound primary HHC through trans diaphragmatic migration, due to small closure was done in the usual fashion and recovery was unevent- leakage [2] in the HHC wall, into the adjacent pericardial cavity to ful. Histopathology confirmed the diagnosis of HC. The preoperative form a PHC. schedule of oral albendazole tablets 400 mg twice daily was contin- In our case, abdominal pain, which was the presenting symptom, ued for 6 months. There is no recurrence during 5 years of followup. was produced by stretching of the hepatic capsule, by the expand- ing HHC in the left lobe of liver and was of recent origin. Pericardial effusion, usually from a leaking PHC, can also be caused by transdi- 3. Discussion aphragmatic rupture of a HHC [2], and produce abdominal pain due to hepatic congestion [1]. But it was not so in our case as both the Cystic echinococcosis (hydatidosis) is a cyclozoonotic infection cysts were intact. The abdominal lump was present for about a year, of cosmopolitan distribution, caused commonly by the larval stages but was ignored by the patient till appearance of pain. Such indif- of tapeworm Echinococcus granulosus. This is endemic in the Indian ference to asymptomatic abdominal lumps is common in the rural subcontinent and is prevalent where livestock is raised in associa- areas of Indian subcontinent and other developing countries. An tion with dogs. HCs are most common in the liver (65%) as it is the increased cardiothoracic ratio in chest X-ray led to final diagnosis site of first filtration of the hexacanth Echinococcus embryos [1,2] of combined HHC and PHC, by echocardiography and computerized after intestinal absorption. Hematogenous spread of the embryos tomogram. which escape hepatic capillary filtration, or protoscolices from mature HHC which may leak into systemic circulation, can pass through the right heart to form HCs in the lungs. Passage of the 4. Conclusion embryos after absorption from intestines through the lymphatic route and thoracic duct [1], and even inhalation of the eggs of con- This case presentation, emphasizes the manifestations of a taminated air in susceptible individuals [3], has also been proposed symptomatic hepatic hydatid cyst along with an associated asymp- to form isolated pulmonary HCs. Pulmonary HCs are found in 25% tomatic pericardial hydatid cyst, diagnosed by radiological imaging. of cases [1,2] and an important source of cardiac HCs. Involvement This is of importance to both physicians and surgeons because of the diaphragm by HC from the adjacent upper lobes of liver and appropriate management can be formulated for a successful clinical transdiaphragmatic migration to thoracic cavity occurs in 0.6–16% outcome. of cases of HHC [2] and infect thoracic organs. Incidence of car- diac HC is rare (0.5–2%) even in countries where hydatid disease is Conflict of interest endemic [1,2]. Origin of the cardiac HC is hematogenous. Embryos may enter the and grow to form intramyocar- There is no conflict of interest. dial cysts [1,2,4]. The commonly affected cardiac chambers are the left ventricle (50–60%) followed by right ventricle (15%), while PHCs comprise 10–15% of these cardiac HCs [1,2]. The intramyocar- Funding dial HCs produce early symptoms as they grow and their life span is shortened due to kinetic pressure exerted by the myocardium The authors declare they have no funding support. [4]. The same kinetic pressure by myocardial contractility favors their passage, usually towards the epicardial surface [4], and less Ethical approval frequently towards the subendocardial region with risk of intracav- itory rupture. Ultimatley, a few of these subepicardial HCs perforate Our research is approved by the Ethics Committee of Medical the epicardium and grow it into a large asymptomatic PHC, in the College Hospitals, Kolkata, West Bengal, India. pericardial space, by stretching the fibrous pericardium [1,2,4]. Par- tial rupture of PHC can produce constrictive pericarditis; whereas, complete rupture can lead to tamponade [1,2,4]. Unruptured PHCs Authors’ contribution at a later stage, can produce pressure effect over the heart, like right ventricular outflow tract or great vessel obstruction [1,4], coronary Each of the authors for preparing this case report, Dr. Kallol artery compression with consequent ischemia [1,2]. Contents of Dasbaksi (Corresponding author), Dr. Suranjan Haldar, Dr. Kaushik these cysts is clear during the early stages [4] and may become Mukherjee and Dr. Plaban Mukherjee have contributed to the study cheesy due to degenerative changes later. In our case the PHC was concept or design, data collection, data analysis or interpretation, localized over the left ventricular aspect and contained approxi- for writing the paper. mately 500 cc of clear fluid without any signs and symptoms of pressure effect produced during its long existence, probably coun- terbalanced by high left ventricular pressure. The PHC could be Consent excised in beating heart in our case due to its favourable antero- lateral location. Concomitant myocardial HC or subendocardial HC Patient’s consent has been obtained to publish this report for would require removal under cardiopulmonary bypass [1,4]. academic interest. PHCs like cardiac HCs are usually found in association with pulmonary HCs because of hematogenous or contiguous spread Guarantor from pulmonary HC [2]. Conversely, cardiac HC may also be the primary source, with metastatic spread to pulmonary and other Dr. Kallol Dasbaksi. remote organs, by hematogenous route [5]. The combination of a mature, large PHC and a HHC, as in our case, can not be explained by the known mechanisms of hematogenous or lymphatic spread Appendix A. Supplementary data from a HHC, without sparing the lungs. Whether the combination is just incidental due to separate primary infestation, by infective Supplementary data associated with this article can be found, in larvae [5] at different times, is open to conjecture. One plausible the online version, at http://dx.doi.org/10.1016/j.ijscr.2015.02.052. CASE REPORT – OPEN ACCESS

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References [4] F. Perez-Gomez, H. Duran, S. Tamames, J.L. Perrote, A. Blanes, Cardiac echinococcosis: clinical picture and complications, Br. Heart J. 35 (12) (1973) [1] H. Yaliniz, A. Tokcan, O.K. Salih, T. Ulus, Surgical treatment of cardiac hydatid 1326–1331. disease: a report of 7 cases, Texas Heart Inst. J. 33 (3) (2006) 333–339. [5] J.V. Deshmane, Multiple pulmonary hydatid cysts, Postgrad. Med. J. 43 (1967) [2] P. Polat, M. Kantarci, F. Alper, S. Suma, M.B. Koruyucu, A. Okur, Hydatid disease 774–778. from head to toe, Radiographics 23 (2003) 475–494. [3] S.E. Golzari, M. Sokouti Pericyst, The outermost layer of hydatid cyst, World J. Gastroenterol. 20 (February (5)) (2014) 1377–1378.

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